Eosinophilic chronic rhinosinusitis and concurrent Kimura’s disease treated with mepolizumab

2021 ◽  
Vol 14 (1) ◽  
pp. e232627
Author(s):  
Jacqueline Ho ◽  
Sophie Walter ◽  
Richard J Harvey
Keyword(s):  
English Language ◽  
Cervical Lymphadenopathy ◽  
Upper Airway ◽  
Neck Region ◽  
Kimura’S Disease ◽  
Inflammatory Disorder ◽  
Head And Neck Region ◽  
Chronic Inflammatory Disorder ◽  
First Case ◽  
Eosinophilic Chronic Rhinosinusitis

Kimura’s disease is a rare, benign, chronic inflammatory disorder characterised by its eosinophilic infiltrate. Patients often present with one or more progressively enlarging subcutaneous lymph nodes in the head and neck region or enlarging salivary glands. We describe the case of a 26-year-old man presenting with severe peripheral eosinophilia and upper airway inflammatory symptoms, who later developed cervical lymphadenopathy and formally diagnosed with Kimura’s disease. Based on our English-language MEDLINE literature search, to our knowledge this is the first case report describing treatment of Kimura’s disease with mepolizumab.

scholarly journals Kimura’s disease: a diagnostic and therapeutic enigma

2018 ◽  
Vol 6 (6) ◽  
pp. 2176
Author(s):  
Vinay Bharat ◽  
Abhishek Gupta ◽  
Rani Bansal ◽  
Priya Gupta ◽  
Mamta Gupta
Keyword(s):  
Lymph Nodes ◽  
Elevated Serum ◽  
Neck Region ◽  
Kimura’S Disease ◽  
Inflammatory Disorder ◽  
Neck Swelling ◽  
Peripheral Eosinophilia ◽  
Head And Neck Region ◽  
Chronic Inflammatory Disorder ◽  
Reactive Hyperplasia

Kimura’s disease is a rare chronic inflammatory disorder present in 2nd and 3rd decade. It has a predilection for head and neck region presenting as a slowly growing painless swelling. It is usually accompanied by peripheral eosinophilia and elevated serum IgE and hence it was initially thought to be of allergic origin. Histologically the lesions are characterized by reactive hyperplasia of lymph nodes, eosinophilic infiltration and increase in postcapillary venules. Authors have reported a male patient with a slowly growing right sided neck swelling which is recurring even after course of steroids and excision done twice at an interval of 6 months. Kimura’s disease although a benign Lymphoid disorder but the incidence of recurrence despite taking treatment is a cause of much concern for the patient.

Oropharyngeal Kimura’s disease: a diagnostic dilemma and therapeutic challenge

2020 ◽  
Vol 13 (10) ◽  
pp. e236366
Author(s):  
Prasanna Kumar Saravanam ◽  
Aishwarya Gajendran ◽  
Nethra Dinakaran ◽  
Dhaarani Jayaraman
Keyword(s):  
Histopathological Examination ◽  
Subcutaneous Tissue ◽  
Neck Region ◽  
Treatment Modalities ◽  
Kimura’S Disease ◽  
Inflammatory Disorder ◽  
First Line ◽  
Diagnostic Dilemma ◽  
Head And Neck Region ◽  
Chronic Inflammatory Disorder

Kimura’s disease is a chronic inflammatory disorder involving subcutaneous tissue and locoregional lymph nodes of head and neck region, characterised by angiolymphoid proliferation. It is typically associated with peripheral eosinophilia, increased serum IgE and lymphadenopathy. Diagnosis is usually based on histopathological examination. Though various treatment modalities have been put forward, surgical management remains the first line of treatment, followed by systemic steroids application. This case report discusses the diagnostic dilemma and therapeutic management of a rare clinical presentation and management of oropharyngeal Kimura’s disease in a young immunocompetent woman.

scholarly journals Rituximab Treatment in a Patient with Kimura Disease and Membranous Nephropathy: Case Report

2021 ◽  
pp. 116-123
Author(s):  
Roald Vissing-Uhre ◽  
Alastair Hansen ◽  
Susanne Frevert ◽  
Ditte Hansen
Keyword(s):  
Case Report ◽  
Membranous Nephropathy ◽  
Neck Region ◽  
Renal Diseases ◽  
Kimura’S Disease ◽  
Inflammatory Disorder ◽  
Kimura Disease ◽  
Chronic Inflammatory Disorder ◽  
Eosinophil Granulocytes ◽  
Anti Cd20

Kimura disease (KD) is a chronic, inflammatory disorder with slowly developing subcutaneous tumor-like swellings, often occurring in the head and neck region. KD is diagnosed based on histology, elevated levels of immunoglobulin type E, and increased peripheral eosinophil granulocytes. KD may coexist with glomerular renal diseases, and this case report is based on a patient with KD-associated membranous nephropathy. Patients with membranous nephropathy without KD have demonstrated responsiveness to treatment with monoclonal anti-CD20 antibodies. This case report is the first to investigate the effect of rituximab treatment in a patient with KD-associated membranous nephropathy. A 30-year-old Italian man living in Denmark was diagnosed with Kimura’s disease based on subcutaneous nodules with eosinophil angiolymphoid hyperplasia. The patient was admitted to the hospital due to nephrotic syndrome. Serology showed eosinophil granulocytosis and negative PLA2-receptor antibody. Renal biopsy showed membranous nephropathy, and the patient was treated with systemic methylprednisolone followed by cyclosporin and then cyclophosphamide with only partial remission. Ultimately, treatment with intravenous rituximab was initiated, which resulted in overall remission and no nephrotic relapses at 30 months of follow-up. Thus, intravenous rituximab effectively decreased proteinuria and prevented nephrotic relapses in a patient with treatment-refractory membranous nephropathy due to KD.

scholarly journals Kimura’s disease: a rare cause of facial mass in a caucasian male patient

2020 ◽  
Vol 6 (4) ◽  
pp. 20200099
Author(s):  
Célia Peixoto Sousa ◽  
Elsa Fonseca ◽  
Bárbara Viamonte ◽  
João Calheiros Lobo ◽  
António Madureira
Keyword(s):  
Immunoglobulin E ◽  
Neck Region ◽  
High Serum ◽  
Kimura’S Disease ◽  
Inflammatory Disorder ◽  
Peripheral Eosinophilia ◽  
Review Of The Literature ◽  
Head And Neck Region ◽  
Caucasian Male ◽  
Subcutaneous Mass

Kimura’s disease is an uncommon inflammatory disorder of unclear aetiology, mainly affecting young Asian descent males among their second and fourth decades of life. The disease typically emerges as a long-standing and painless subcutaneous mass lesion in the head and neck region, frequently associated with swelling of major salivary glands, particularly the parotid gland, and regional lymphadenopathy. Peripheral eosinophilia and high serum immunoglobulin E are also characteristic findings. We report a case and describe the imaging and pathological features of the disease in a 19-year-old Caucasian male, with review of the literature.

scholarly journals Kimura Disease of the Parotid Gland: Management Approach

2022 ◽  
Vol 4 (1) ◽  
pp. 01-03
Author(s):  
Hafeza MS ◽  
Vanitha Palanisamy
Keyword(s):  
Parotid Gland ◽  
Frozen Section ◽  
Neck Region ◽  
Unknown Etiology ◽  
Management Approach ◽  
Kimura’S Disease ◽  
Inflammatory Disorder ◽  
Chronic Inflammatory Disorder ◽  
Parotid Neoplasm ◽  
The Right

Here we present a 43-years old Malay man with Kimura’s disease of the right parotid gland. Asian men have a predisposition for this chronic inflammatory disorder of unknown etiology mostly presenting at the head and neck region. The management in dealing with this patient is not straight forward as the presentation mimics a subcutaneous tumor-like mass in which main differential diagnosis includes lymphoproliferative disorders and parotid neoplasm. Moreover, cytological investigation is inadequate, and this necessitates the use intra-operative frozen section prior to tumor debulking. Without proper investigation, misdiagnosis can occur and potentially expose the patient to unnecessary extensive surgical procedures. Here we discuss step by step approach in managing this patient.

Kimura Disease: A Rare Cause of Head and Neck Tumour in Children- A Case Report

2021 ◽  
Vol 17 ◽  
Author(s):  
Mohammad Hanafiah Kreah ◽  
Mohd Nadzri Misni ◽  
Nazimah Ab Mumin
Keyword(s):  
Head And Neck ◽  
Cervical Lymphadenopathy ◽  
Neck Region ◽  
Kimura’S Disease ◽  
Parotid Glands ◽  
Kimura Disease ◽  
Case Presentation ◽  
Head And Neck Region ◽  
Eyelid Swelling ◽  
Nodular Lesions

Background: Kimura disease is a rare inflammatory condition classically manifested as painless subcutaneous nodules in the head and neck region and associated with regional cervical lymphadenopathy and salivary gland involvement. Objective: The purpose of this report is to illustrate the diagnostic difficulty due to its rarity and non-specific clinical presentation. Case Presentation: We present a case of Kimura disease with bilateral eyelid swelling, parotid involvement, and cervical lymphadenopathy in a young boy. Computed Tomography (CT) showed ill-defined enhancing masses in both upper eyelids with lacrimal gland infiltration, multiple nodular lesions in both parotid glands, and bilateral enlargement of cervical nodes. After several multidisciplinary discussions and trials of medications, Kimura's disease was finally confirmed by histology examination. Conclusion: In conclusion, a rare disease such as Kimura must be considered earlier if the patient is not responding to the treatment.

scholarly journals Malakoplakia of the parotid gland: a case report and review of localised malakoplakia of the head and neck

2019 ◽  
Vol 101 (5) ◽  
pp. 309-312
Author(s):  
C Samian ◽  
S Ghaffar ◽  
V Nandapalan ◽  
S Santosh
Keyword(s):  
Salivary Gland ◽  
Case Report ◽  
Parotid Gland ◽  
Head And Neck ◽  
World Literature ◽  
Neck Region ◽  
Submandibular Salivary Gland ◽  
Inflammatory Disorder ◽  
Head And Neck Region ◽  
Chronic Inflammatory Disorder

Introduction Malakoplakia is a rare chronic inflammatory disorder, predominantly affecting the urinary tract. In the head and neck region, it is very rare and may confuse the clinicians during investigations, as features may mimic malignancy. Materials and methods We report a case of malakoplakia involving the parotid gland and review of the reported cases of malakoplakia in head and neck region. Results Histologically, this is the first classic case report of malakoplakia involving the parotid gland in the world literature. A total of 49 cases have been reported in the head and neck region; 38.7% of these are cutaneous. In soft tissue, the tongue is the most common site. Salivary gland involvement is very rare. Previously, submandibular salivary gland involvement has been reported. Conclusion A possibility of malakoplakia should be considered as a differential diagnosis in patients with enlarged head and neck masses. Histology is essential to diagnose this benign inflammatory disorder and to differentiate from a malignant process.

scholarly journals Kimura Disease as a Rare Cause of Proptosis: A Case Report

2021 ◽  
Vol 16 (2) ◽  
pp. 259-265
Author(s):  
V Sha Kri Eh Dam ◽  
Irfan Mohamad ◽  
Evelyn Li Min Tai ◽  
Adil Hussein ◽  
Khairil Amir Sayuti ◽  
...  
Keyword(s):  
Head And Neck ◽  
Neck Region ◽  
Oral Prednisolone ◽  
Lymph Node Involvement ◽  
High Recurrence Rate ◽  
Inflammatory Disorder ◽  
Kimura Disease ◽  
Head And Neck Region ◽  
Chronic Inflammatory Disorder ◽  
Middle Aged Adult

Kimura disease (KD) is a rare chronic inflammatory disorder of unknown aetiology that primarily affects the head and neck region with lymph node involvement. Young to middle-aged adult Asian males are predominantly affected. The most common presentation is painless subcutaneous swelling in the head and neck region, while proptosis or orbital involvement is very rarely reported. KD shares some features with other inflammatory and neoplastic disorders, including lymphoma; thus, investigations to confirm the diagnosis should not be delayed. Systemic corticosteroids are commonly used to treat KD and show an excellent response; however, the optimal treatment is still uncertain, and KD has a high recurrence rate. We describe the case of a patient with KD who presented with proptosis and post-auricular swelling, which responded well to oral prednisolone treatment.

scholarly journals Kimura’s Disease: A Rare Cause of Unilateral Tonsillar Enlargement

2021 ◽  
Vol 2021 ◽  
pp. 1-4
Author(s):  
Prakash Khanal ◽  
Agya Shrestha
Keyword(s):  
Histopathological Examination ◽  
Cervical Lymphadenopathy ◽  
Neck Region ◽  
Rare Condition ◽  
Surgical Excision ◽  
Kimura’S Disease ◽  
Inflammatory Disorder ◽  
Serum Ige ◽  
History Of ◽  
High Index Of Suspicion

Introduction. Kimura’s disease is a rare inflammatory disorder of unknown cause, commonly seen in young Asian males. Case Report. A 61-year-old male patient presented with a history of right tonsillar mass and cervical lymphadenopathy. The patient underwent hematological investigation and imaging followed by resection of tonsillar mass. Based on histopathological and subsequent immunohistochemistry reports, the case was diagnosed as Kimura’s disease of the tonsil. Discussion. Kimura’s disease commonly presents as painless subcutaneous masses in the head and neck region or cervical lymphadenopathy. Kimura’s disease presenting as a tonsillar mass is a very rare condition. Patients usually have peripheral eosinophilia and elevated levels of serum IgE. The diagnosis is based on the clinical and histopathologic findings in a biopsy of the mass and/or lymph node along with elevated peripheral eosinophil and serum IgE level. Conclusion. The clinical presentation of Kimura’s disease is highly variable. Kimura’s disease should be considered as a differential diagnosis in patients presenting with a tonsillar mass. A high index of suspicion along with histopathological examination helps in the early diagnosis and management. Surgical excision is the treatment of choice.

scholarly journals Kimura’s disease as an uncommon cause of post auricular swelling

2020 ◽  
Vol 6 (9) ◽  
pp. 1737
Author(s):  
Chandre Gowda Bendiganahalli Venkate Gowda ◽  
Madhuri Gandham
Keyword(s):  
Immunoglobulin E ◽  
Neck Region ◽  
Unknown Etiology ◽  
Kimura’S Disease ◽  
Histopathological Diagnosis ◽  
Inflammatory Disorder ◽  
Chronic Inflammatory Disorder ◽  
Tissue Eosinophilia ◽  
Common Causes ◽  
Rule Out

<p>Kimura’s disease (KD) is a rare chronic inflammatory disorder of unknown etiology, primarily seen in young Asian males. In India, only 200 cases have been reported worldwide since its histopathological diagnosis. The disease is characterized by painless subcutaneous swelling in head &amp; neck region, blood and tissue eosinophilia and raised Immunoglobulin E (IgE) levels. A systematic multidisciplinary approach is mandatory to rule out the other common causes of post auricular lymphadenopathy. The diagnosis of KD can be difficult and misleading and patients with this disease are often evaluated using avoidable procedures by just not being aware of KD. Here, we present a case of a 20 year old male who presented with nodular swellings in the bilateral post auricular region. The diagnosis of KD was done based on characteristic histopathologic finding in conjunction with peripheral eosinophilia.</p>

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